Alenezi Ahmad F, Jahrami Haitham, Kanber Yonca, Bessissow Talat
Department of Medicine, McGill University, 3605 Rue de La Montagne, Montreal H3G 2M1, Quebec, Canada.
Department of Government Hospitals, Ministry of Health, Road 2909, Salmanyia, Manama, Bahrain.
Case Rep Gastrointest Med. 2025 Mar 9;2025:1636142. doi: 10.1155/crgm/1636142. eCollection 2025.
Perineurioma is a rare benign peripheral nerve sheath tumor that can arise in various body locations. In the gastrointestinal (GI) tract, perineuriomas are uncommon and have only been reported in case reports and case series. In addition, a new classification suggests reclassifying benign fibroblastic polyps as perineurioma when they show positive markers of perineurial differentiation. This study aims to enhance understanding of GI tract perineuriomas by presenting a new case and conducting a systematic literature review. We described a new case of colonic perineurioma and systematically reviewed all case reports and case series on GI perineuriomas and benign fibroblastic polyps with perineurial markers. We searched ScienceDirect, PubMed/MEDLINE, and Web of Science up to May 2024. A total of 148 cases were analyzed, and most of the cases were published in the last decade (2014-2024). The majority were females (59.46%), with a mean age of 51 years (standard deviation [SD] ±14.87). Most GI perineuriomas (87.5%) were in the distal colon, predominantly in the sigmoid/rectosigmoid (56%) and rectum (14%). Outside the colon, the stomach was the most affected site (7 of 10 cases), with fewer cases in the small intestine and esophagus. The two most commonly performed stains were for epithelial membrane antigen (EMA) and glucose transporter 1 (GLUT-1), at 75% and 56% of cases, respectively. Noncolonic perineuriomas were generally larger and more symptomatic than colonic ones. Submucosal polyps were more likely symptomatic than mucosal polyps. Perineurioma in the GI tract is a rare benign polyp mainly identified in the distal colon. Its rarity and limited follow-up data restrict our understanding of recurrence rates. We recommend reporting uncommon polyp locations, detailing polyp morphologies, and using at least two markers for classification.
神经束膜瘤是一种罕见的良性周围神经鞘瘤，可发生于身体的各个部位。在胃肠道（GI）中，神经束膜瘤并不常见，仅在病例报告和病例系列中有报道。此外，一种新的分类方法建议，当良性纤维母细胞性息肉显示神经束膜分化的阳性标志物时，将其重新分类为神经束膜瘤。本研究旨在通过报告一例新病例并进行系统的文献综述，加深对胃肠道神经束膜瘤的了解。我们描述了一例新的结肠神经束膜瘤病例，并系统回顾了所有关于胃肠道神经束膜瘤和具有神经束膜标志物的良性纤维母细胞性息肉的病例报告和病例系列。我们检索了截至2024年5月的ScienceDirect、PubMed/MEDLINE和Web of Science。共分析了148例病例，大多数病例发表于过去十年（2014 - 2024年）。大多数为女性（59.46%），平均年龄为51岁（标准差[SD]±14.87）。大多数胃肠道神经束膜瘤（87.5%）位于结肠远端，主要在乙状结肠/直肠乙状结肠交界处（56%）和直肠（14%）。在结肠外，胃是最常受累的部位（10例中有7例），小肠和食管的病例较少。最常用的两种染色分别是上皮膜抗原（EMA）和葡萄糖转运蛋白1（GLUT - 1），分别占病例的75%和56%。非结肠神经束膜瘤通常比结肠神经束膜瘤更大且症状更明显。黏膜下息肉比黏膜息肉更易出现症状。胃肠道神经束膜瘤是一种罕见 的良性息肉，主要见于结肠远端。其罕见性和有限的随访数据限制了我们对复发率的了解。我们建议报告罕见的息肉位置，详细描述息肉形态，并使用至少两种标志物进行分类。
